This paper desceibes an autopsy case of a new born male infant with complex malformations of the rectum and the urinary system. The rectal abnormality was of a form of true rectal atresia without accompaniment of fistula formation. The anomalies of the urinary system consisted of (1) a stenosis of the bladder neck due to sub-mucosal fibrosis and (2) a marked degree of primary muscular hypertrophy of the bladder wall. A development mechanism of the true rectal atresia was briefly considered and a possibility of its high coincidence with the anomalies of lower urinary tracts was discussed.